Letter to the Editor
Linear scleroderma en coup de sabre and epilepsy: Presentation of a case in a child
Esclerodermia lineal en golpe de sable y epilepsia. A propósito de un caso infantil
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Garófalo Gómez, L. Novoa López, A.M. Gómez García, M. Méndez Méndez" "autores" => array:4 [ 0 => array:4 [ "nombre" => "N." "apellidos" => "Garófalo Gómez" "email" => array:1 [ 0 => "nicogaro@infomed.sld.cu" ] "referencia" => array:2 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">¿</span>" "identificador" => "cor0005" ] ] ] 1 => array:3 [ "nombre" => "L." "apellidos" => "Novoa López" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] ] ] 2 => array:3 [ "nombre" => "A.M." "apellidos" => "Gómez García" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] ] ] 3 => array:3 [ "nombre" => "M." "apellidos" => "Méndez Méndez" "referencia" => array:1 [ 0 => array:2 [ "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] ] ] "afiliaciones" => array:3 [ 0 => array:3 [ "entidad" => "Departamento de Neuropediatría del Instituto de Neurología y Neurocirugía de Cuba, La Habana, Cuba" "etiqueta" => "<span class="elsevierStyleSup">a</span>" "identificador" => "aff0005" ] 1 => array:3 [ "entidad" => "Universidad de Ciencias Médicas de La Habana, La Habana, Cuba" "etiqueta" => "<span class="elsevierStyleSup">b</span>" "identificador" => "aff0010" ] 2 => array:3 [ "entidad" => "Servicio Nacional de Reumatología Pediátrica, Hospital Universitario Pedro Borrás Astorga, La Habana, Cuba" "etiqueta" => "<span class="elsevierStyleSup">c</span>" "identificador" => "aff0015" ] ] "correspondencia" => array:1 [ 0 => array:3 [ "identificador" => "cor0005" "etiqueta" => "⁎" "correspondencia" => "Corresponding author." ] ] ] ] "titulosAlternativos" => array:1 [ "es" => array:1 [ "titulo" => "Esclerodermia lineal en golpe de sable y epilepsia. A propósito de un caso infantil" ] ] "resumenGrafico" => array:2 [ "original" => 0 "multimedia" => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 642 "Ancho" => 1301 "Tamanyo" => 81211 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Brain CT: (A) At 4 years of age and (B) at 7 years of age.</p>" ] ] ] "textoCompleto" => "<span class="elsevierStyleSections"><p id="par0005" class="elsevierStylePara elsevierViewall">Localised scleroderma (LS) is a rare autoimmune disorder that primarily affects the skin and may also affect underlying fatty, muscle, or bone tissue.<a class="elsevierStyleCrossRef" href="#bib0005"><span class="elsevierStyleSup">1</span></a> The estimated prevalence of this disease is fewer than 3 cases in 100<span class="elsevierStyleHsp" style=""></span>000 inhabitants.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0010" class="elsevierStylePara elsevierViewall">LS affects the skin almost exclusively, and with rare exceptions, does not injure internal organs. It is categorised into 5 subtypes: circumscribed morphea, linear scleroderma, generalised morphea, pansclerotic morphea, and a mixed subtype.<a class="elsevierStyleCrossRef" href="#bib0015"><span class="elsevierStyleSup">3</span></a></p><p id="par0015" class="elsevierStylePara elsevierViewall">Linear scleroderma en coup de sabre (LSCS) is a descriptive term indicating the presence of LS on the frontoparietal face and scalp. This uncommon form of LS mainly occurs in paediatric patients; neurological symptoms, especially epilepsy, are also relatively common in these patients.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,4</span></a></p><p id="par0020" class="elsevierStylePara elsevierViewall">We present the case of a 7-year-old male whose state of health was normal until the age of 20 months when he began to show hyperpigmented lesions on the left side of his face (forehead and nose) with progressive atrophy of the skin and underlying tissue in that region. LSCS was diagnosed based on the clinical profile with pathognomonic signs of the linear subtype of scleroderma, and on the criteria for classifying systemic juvenile sclerosis<a class="elsevierStyleCrossRefs" href="#bib0025"><span class="elsevierStyleSup">5,6</span></a> (<a class="elsevierStyleCrossRef" href="#fig0005">Fig. 1</a>).</p><elsevierMultimedia ident="fig0005"></elsevierMultimedia><p id="par0025" class="elsevierStylePara elsevierViewall">Six months after onset of the illness, the patient began to experience episodes of right-sided deviation of the eyes and head with episodes of altered consciousness lasting a few seconds, followed by rapid complete recovery (1–2<span class="elsevierStyleHsp" style=""></span>min). He was diagnosed with focal symptomatic epilepsy and started carbamazepine treatment.</p><p id="par0030" class="elsevierStylePara elsevierViewall">When the child was 5 years old, he described vision loss at the onset of seizures. Clobazam was added to his treatment programme, and seizures remain controlled to date.</p><p id="par0035" class="elsevierStylePara elsevierViewall">The neuropsychological study performed when the patient was 7 years old showed an intellectual level in the lower normal range (WISC-R: performance IQ 63; verbal IQ 84; full scale IQ 71).</p><p id="par0040" class="elsevierStylePara elsevierViewall">Cranial CT scans completed when the patient was 4 and 7 years old showed an increase in calcification in the left brain hemisphere and a hypodense area in the head of the left caudate nucleus that suggested an old cerebral infarct (<a class="elsevierStyleCrossRef" href="#fig0010">Fig. 2</a>).</p><elsevierMultimedia ident="fig0010"></elsevierMultimedia><p id="par0045" class="elsevierStylePara elsevierViewall">Multiple neurological manifestations may be associated with LSCS; of these, focal epileptic seizures are the most common.<a class="elsevierStyleCrossRefs" href="#bib0035"><span class="elsevierStyleSup">7–13</span></a></p><p id="par0050" class="elsevierStylePara elsevierViewall">Changes detected in CT studies tend to be ipsilateral to skin lesions and may include the following: thinning or depression of the external diploë, focal or hemispheric cerebral atrophy, and intracranial calcifications.<a class="elsevierStyleCrossRefs" href="#bib0010"><span class="elsevierStyleSup">2,6,14</span></a></p><p id="par0055" class="elsevierStylePara elsevierViewall">Cerebral lesions caused by infarct are rare in linear scleroderma, so the presence of a cerebral infarct in our patient deserves mention. There are few reports of cerebral infarcts occurring in patients with LSCS.<a class="elsevierStyleCrossRef" href="#bib0010"><span class="elsevierStyleSup">2</span></a></p><p id="par0060" class="elsevierStylePara elsevierViewall">Calcified intracranial lesions may be produced by an inflammatory process in cerebral blood vessels. Interestingly enough, these lesions arise on the same side as the skin lesions, and there is still no reliable scientific explanation for this phenomenon. The autoimmune hypothesis has the best evidence in its favour. It is based on reports of findings from cerebral biopsies showing inflammatory changes in the cerebral parenchyma, and sometimes in blood vessels and the meninges as well.<a class="elsevierStyleCrossRef" href="#bib0075"><span class="elsevierStyleSup">15</span></a></p><p id="par0065" class="elsevierStylePara elsevierViewall">In the case we present, we find evidence that the disease remains active; intracranial calcifications have continued growing slowly and gradually, despite use of correctly dosed immunosuppressants. Even considering the course of the disease and the presence of extracutaneous neurological signs, the patient has never met all the diagnostic criteria for systemic juvenile sclerosis.<a class="elsevierStyleCrossRef" href="#bib0025"><span class="elsevierStyleSup">5</span></a></p></span>" "pdfFichero" => "main.pdf" "tienePdf" => true "NotaPie" => array:1 [ 0 => array:2 [ "etiqueta" => "☆" "nota" => "<p class="elsevierStyleNotepara">Please cite this article as: Garófalo Gómez N, et al. Esclerodermia lineal en golpe de sable y epilepsia. A propósito de un caso infantil. Neurología. 2012;27:449–51.</p>" ] ] "multimedia" => array:2 [ 0 => array:7 [ "identificador" => "fig0005" "etiqueta" => "Figure 1" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr1.jpeg" "Alto" => 729 "Ancho" => 799 "Tamanyo" => 84557 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0005" class="elsevierStyleSimplePara elsevierViewall">Patient photo in which we observe the typical left frontoparietal lesion “en coup de sable”.</p>" ] ] 1 => array:7 [ "identificador" => "fig0010" "etiqueta" => "Figure 2" "tipo" => "MULTIMEDIAFIGURA" "mostrarFloat" => true "mostrarDisplay" => false "figura" => array:1 [ 0 => array:4 [ "imagen" => "gr2.jpeg" "Alto" => 642 "Ancho" => 1301 "Tamanyo" => 81211 ] ] "descripcion" => array:1 [ "en" => "<p id="spar0010" class="elsevierStyleSimplePara elsevierViewall">Brain CT: (A) At 4 years of age and (B) at 7 years of age.</p>" ] ] ] "bibliografia" => array:2 [ "titulo" => "References" "seccion" => array:1 [ 0 => array:2 [ "identificador" => "bibs0005" "bibliografiaReferencia" => array:15 [ 0 => array:3 [ "identificador" => "bib0005" "etiqueta" => "1" "referencia" => array:1 [ 0 => array:2 [ "contribucion" => array:1 [ 0 => array:2 [ "titulo" => "AWMF guideline n.° 013/066. 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| Year/Month | Html | Total | |
|---|---|---|---|
| 2024 November | 7 | 2 | 9 |
| 2024 October | 80 | 4 | 84 |
| 2024 September | 82 | 11 | 93 |
| 2024 August | 91 | 7 | 98 |
| 2024 July | 78 | 4 | 82 |
| 2024 June | 72 | 2 | 74 |
| 2024 May | 93 | 4 | 97 |
| 2024 April | 80 | 4 | 84 |
| 2024 March | 98 | 4 | 102 |
| 2024 February | 108 | 11 | 119 |
| 2024 January | 129 | 6 | 135 |
| 2023 December | 113 | 12 | 125 |
| 2023 November | 180 | 10 | 190 |
| 2023 October | 179 | 18 | 197 |
| 2023 September | 80 | 6 | 86 |
| 2023 August | 129 | 5 | 134 |
| 2023 July | 157 | 9 | 166 |
| 2023 June | 97 | 1 | 98 |
| 2023 May | 123 | 8 | 131 |
| 2023 April | 250 | 4 | 254 |
| 2023 March | 290 | 1 | 291 |
| 2023 February | 211 | 4 | 215 |
| 2023 January | 146 | 17 | 163 |
| 2022 December | 98 | 4 | 102 |
| 2022 November | 131 | 8 | 139 |
| 2022 October | 92 | 10 | 102 |
| 2022 September | 118 | 10 | 128 |
| 2022 August | 92 | 9 | 101 |
| 2022 July | 68 | 9 | 77 |
| 2022 June | 82 | 10 | 92 |
| 2022 May | 59 | 8 | 67 |
| 2022 April | 57 | 7 | 64 |
| 2022 March | 111 | 7 | 118 |
| 2022 February | 106 | 4 | 110 |
| 2022 January | 126 | 10 | 136 |
| 2021 December | 74 | 15 | 89 |
| 2021 November | 77 | 5 | 82 |
| 2021 October | 115 | 11 | 126 |
| 2021 September | 105 | 13 | 118 |
| 2021 August | 84 | 5 | 89 |
| 2021 July | 69 | 8 | 77 |
| 2021 June | 89 | 10 | 99 |
| 2021 May | 122 | 12 | 134 |
| 2021 April | 234 | 21 | 255 |
| 2021 March | 127 | 9 | 136 |
| 2021 February | 87 | 11 | 98 |
| 2021 January | 101 | 17 | 118 |
| 2020 December | 89 | 12 | 101 |
| 2020 November | 63 | 10 | 73 |
| 2020 October | 43 | 5 | 48 |
| 2020 September | 63 | 5 | 68 |
| 2020 August | 55 | 12 | 67 |
| 2020 July | 48 | 14 | 62 |
| 2020 June | 44 | 4 | 48 |
| 2020 May | 53 | 8 | 61 |
| 2020 April | 44 | 4 | 48 |
| 2020 March | 45 | 1 | 46 |
| 2020 February | 43 | 1 | 44 |
| 2020 January | 50 | 5 | 55 |
| 2019 December | 67 | 5 | 72 |
| 2019 November | 46 | 4 | 50 |
| 2019 October | 45 | 6 | 51 |
| 2019 September | 48 | 3 | 51 |
| 2019 August | 34 | 2 | 36 |
| 2019 July | 36 | 12 | 48 |
| 2019 June | 109 | 27 | 136 |
| 2019 May | 293 | 28 | 321 |
| 2019 April | 146 | 13 | 159 |
| 2019 March | 24 | 7 | 31 |
| 2019 February | 35 | 11 | 46 |
| 2019 January | 45 | 5 | 50 |
| 2018 December | 24 | 4 | 28 |
| 2018 November | 41 | 11 | 52 |
| 2018 October | 81 | 9 | 90 |
| 2018 September | 93 | 9 | 102 |
| 2018 August | 26 | 5 | 31 |
| 2018 July | 19 | 7 | 26 |
| 2018 June | 16 | 7 | 23 |
| 2018 May | 31 | 8 | 39 |
| 2018 April | 21 | 2 | 23 |
| 2018 March | 19 | 2 | 21 |
| 2018 February | 10 | 0 | 10 |
| 2018 January | 22 | 4 | 26 |
| 2017 December | 10 | 3 | 13 |
| 2017 November | 18 | 8 | 26 |
| 2017 October | 17 | 1 | 18 |
| 2017 September | 26 | 8 | 34 |
| 2017 August | 23 | 8 | 31 |
| 2017 July | 24 | 11 | 35 |
| 2017 June | 23 | 6 | 29 |
| 2017 May | 32 | 12 | 44 |
| 2017 April | 23 | 7 | 30 |
| 2017 March | 21 | 9 | 30 |
| 2017 February | 24 | 8 | 32 |
| 2017 January | 24 | 1 | 25 |
| 2016 December | 27 | 13 | 40 |
| 2016 November | 34 | 12 | 46 |
| 2016 October | 40 | 15 | 55 |
| 2016 September | 38 | 11 | 49 |
| 2016 August | 46 | 9 | 55 |
| 2016 July | 16 | 2 | 18 |
| 2016 June | 34 | 9 | 43 |
| 2016 May | 29 | 14 | 43 |
| 2016 April | 32 | 11 | 43 |
| 2016 March | 50 | 21 | 71 |
| 2016 February | 41 | 19 | 60 |
| 2016 January | 25 | 13 | 38 |
| 2015 December | 26 | 12 | 38 |
| 2015 November | 27 | 19 | 46 |
| 2015 October | 41 | 20 | 61 |
| 2015 September | 33 | 13 | 46 |
| 2015 August | 48 | 11 | 59 |
| 2015 July | 98 | 10 | 108 |
| 2015 June | 40 | 2 | 42 |
| 2015 May | 47 | 12 | 59 |
| 2015 April | 43 | 12 | 55 |
| 2015 March | 50 | 14 | 64 |
| 2015 February | 38 | 11 | 49 |
| 2015 January | 28 | 6 | 34 |
| 2014 December | 31 | 10 | 41 |
| 2014 November | 27 | 2 | 29 |
| 2014 October | 20 | 2 | 22 |
| 2014 September | 40 | 0 | 40 |
| 2014 August | 44 | 8 | 52 |
| 2014 July | 43 | 6 | 49 |
| 2014 June | 27 | 2 | 29 |
| 2014 May | 22 | 4 | 26 |
| 2014 April | 35 | 3 | 38 |
| 2014 March | 19 | 5 | 24 |
| 2014 February | 21 | 0 | 21 |
| 2014 January | 19 | 4 | 23 |
| 2013 December | 31 | 2 | 33 |
| 2013 November | 43 | 4 | 47 |
| 2013 October | 50 | 10 | 60 |
| 2013 September | 43 | 3 | 46 |
| 2013 August | 42 | 4 | 46 |
| 2013 July | 21 | 1 | 22 |
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